Literature DB >> 2722184

Molecular defects of type III procollagen in Ehlers-Danlos syndrome type IV.

A Superti-Furga1, B Steinmann, F Ramirez, P H Byers.   

Abstract

Fibroblasts from most patients with Ehlers-Danlos syndrome (EDS) type IV, a disorder characterized by fragility of skin, blood vessels, and internal organs, secrete reduced amounts of type III procollagen. In 7 of 8 cell strains analyzed, we found evidence of structural defects in half of the type III procollagen chains synthesized, such as deletions or bona fide amino acid substitutions, which cause delayed formation and destabilization of the collagen triple helix and, as a consequence, reduced secretion of the molecule. The data suggest that EDS type IV is often caused by heterozygosity for mutations at the COL3A1 locus, which affect the structure of type III procollagen. The triple-helical region of the molecule, like the homologous region of type I procollagen, appears to be particularly vulnerable.

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Year:  1989        PMID: 2722184     DOI: 10.1007/BF00284038

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  22 in total

1.  Ehlers-Danlos syndrome type IV: a subset of patients distinguished by low serum levels of the amino-terminal propeptide of type III procollagen.

Authors:  B Steinmann; A Superti-Furga; H I Joller-Jemelka; G Cetta; P H Byers
Journal:  Am J Med Genet       Date:  1989-09

2.  Inheritance of Ehlers-Danlos type IV syndrome.

Authors:  F M Pope; G R Martin; V A McKusick
Journal:  J Med Genet       Date:  1977-06       Impact factor: 6.318

Review 3.  Imperfect collagenesis in osteogenesis imperfecta. The consequences of cysteine-glycine substitutions upon collagen structure and metabolism.

Authors:  B Steinmann; A Superti-Furga; P M Royce
Journal:  Ann N Y Acad Sci       Date:  1988       Impact factor: 5.691

4.  Impaired secretion of type III procollagen in Ehlers-Danlos syndrome type IV fibroblasts: correction of the defect by incubation at reduced temperature and demonstration of subtle alterations in the triple-helical region of the molecule.

Authors:  A Superti-Furga; B Steinmann
Journal:  Biochem Biophys Res Commun       Date:  1988-01-15       Impact factor: 3.575

Review 5.  Human collagens: biochemical, molecular and genetic features in normal and diseased states.

Authors:  F Ramirez; F O Sangiorgi; P Tsipouras
Journal:  Horiz Biochem Biophys       Date:  1986

Review 6.  Molecular basis of inherited disorders of collagen biosynthesis: implications for prenatal diagnosis.

Authors:  P H Byers; R J Wenstrup; J F Bonadio; B Starman; D H Cohn
Journal:  Curr Probl Dermatol       Date:  1987

7.  Ehlers-Danlos syndrome type IV: a multi-exon deletion in one of the two COL3A1 alleles affecting structure, stability, and processing of type III procollagen.

Authors:  A Superti-Furga; E Gugler; R Gitzelmann; B Steinmann
Journal:  J Biol Chem       Date:  1988-05-05       Impact factor: 5.157

8.  Altered secretion of type III procollagen in a form of type IV Ehlers-Danlos syndrome. Biochemical studies in cultured fibroblasts.

Authors:  P H Byers; K A Holbrook; G S Barsh; L T Smith; P Bornstein
Journal:  Lab Invest       Date:  1981-04       Impact factor: 5.662

9.  Synthesis of an altered type III procollagen in a patient with type IV Ehlers-Danlos syndrome. A structural change in the alpha 1(III) chain which makes the protein more susceptible to proteinases.

Authors:  C A Stolle; R E Pyeritz; J C Myers; D J Prockop
Journal:  J Biol Chem       Date:  1985-02-10       Impact factor: 5.157

10.  Patients with Ehlers-Danlos syndrome type IV lack type III collagen.

Authors:  F M Pope; G R Martin; J R Lichtenstein; R Penttinen; B Gerson; D W Rowe; V A McKusick
Journal:  Proc Natl Acad Sci U S A       Date:  1975-04       Impact factor: 11.205

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  27 in total

1.  A 27-bp deletion from one allele of the type III collagen gene (COL3A1) in a large family with Ehlers-Danlos syndrome type IV.

Authors:  A J Richards; J C Lloyd; P Narcisi; P N Ward; A C Nicholls; A De Paepe; F M Pope
Journal:  Hum Genet       Date:  1992-01       Impact factor: 4.132

2.  Substitution of aspartate for glycine 1018 in the type III procollagen (COL3A1) gene causes type IV Ehlers-Danlos syndrome: the mutated allele is present in most blood leukocytes of the asymptomatic and mosaic mother.

Authors:  S Kontusaari; G Tromp; H Kuivaniemi; C Stolle; F M Pope; D J Prockop
Journal:  Am J Hum Genet       Date:  1992-09       Impact factor: 11.025

Review 3.  Genetic causes of aortic aneurysms. Unlearning at least part of what the textbooks say.

Authors:  H Kuivaniemi; G Tromp; D J Prockop
Journal:  J Clin Invest       Date:  1991-11       Impact factor: 14.808

Review 4.  Osteogenesis imperfecta: translation of mutation to phenotype.

Authors:  P H Byers; G A Wallis; M C Willing
Journal:  J Med Genet       Date:  1991-07       Impact factor: 6.318

5.  Linkage analysis of five fibrillar collagen loci in a large French Marfan syndrome family.

Authors:  C Boileau; G Jondeau; C Bonaiti; M Coulon; G Delorme; O Dubourg; J P Bourdarias; C Junien
Journal:  J Med Genet       Date:  1990-02       Impact factor: 6.318

Review 6.  Loeys-Dietz syndrome: cardiovascular, neuroradiological and musculoskeletal imaging findings.

Authors:  Vivek B Kalra; John W Gilbert; Ajay Malhotra
Journal:  Pediatr Radiol       Date:  2011-07-23

7.  A dimorphic Alu Sb-like insertion in COL3A1 is ethnic-specific.

Authors:  D M Milewicz; P H Byers; J Reveille; A L Hughes; M Duvic
Journal:  J Mol Evol       Date:  1996-02       Impact factor: 2.395

8.  The deletion of six amino acids at the C-terminus of the alpha 1 (II) chain causes overmodification of type II and type XI collagen: further evidence for the association between small deletions in COL2A1 and Kniest dysplasia.

Authors:  A Winterpacht; A Superti-Furga; U Schwarze; H Stöss; B Steinmann; J Spranger; B Zabel
Journal:  J Med Genet       Date:  1996-08       Impact factor: 6.318

9.  Clinical Validity of Genes for Heritable Thoracic Aortic Aneurysm and Dissection.

Authors:  Marjolijn Renard; Catherine Francis; Rajarshi Ghosh; Alan F Scott; P Dane Witmer; Lesley C Adès; Gregor U Andelfinger; Pauline Arnaud; Catherine Boileau; Bert L Callewaert; Dongchuan Guo; Nadine Hanna; Mark E Lindsay; Hiroko Morisaki; Takayuki Morisaki; Nicholas Pachter; Leema Robert; Lut Van Laer; Harry C Dietz; Bart L Loeys; Dianna M Milewicz; Julie De Backer
Journal:  J Am Coll Cardiol       Date:  2018-08-07       Impact factor: 24.094

10.  Inheritance of an RNA splicing mutation (G+ 1 IVS20) in the type III procollagen gene (COL3A1) in a family having aortic aneurysms and easy bruisability: phenotypic overlap between familial arterial aneurysms and Ehlers-Danlos syndrome type IV.

Authors:  S Kontusaari; G Tromp; H Kuivaniemi; R L Ladda; D J Prockop
Journal:  Am J Hum Genet       Date:  1990-07       Impact factor: 11.025

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